Infertility and hepatoblastoma: A report from the Children's Oncology Group (COG)

Puumala S.E., Feusner J., Malogolowkin M.H., Krailo M.D., Ross Julia A., Spector L.G.

Research output: Contribution to conferencePaper


Infertility treatment is increasing rapidly in the U.S. A subset of infertility treatment, assisted reproductive technology (ART), has increased 150% between 1996 and 2005 and now accounts for over 1% of all births in the U.S. Parental infertility treatment has been hypothesized to be a risk factor for childhood cancer through potential effects on imprinting and methylation, but few studies have examined treatment as a risk factor for hepatoblastoma (HB). One recent study found a nine-fold increase in HB risk for those with reported or inferred parental infertility treatment. We examined case data from a case-control study of HB conducted through the Children's Oncology Group (COG). Only case data was used since the control series is not yet complete. Proportions of cases whose parents had used infertility services and exact 95% confidence intervals (CI) were constructed and compared to national data. National data were obtained from the 2002 National Survey of Family Growth (NSFG), the ART Surveillance Report 2002-2006, and the National Center for Health Statistics birth data 2002-2006. We also examined the relationship between Beckwith-Wiedemann Syndrome (BWS) and ART in our data set. We included cases of 312 cases HB diagnosed in the year 2000 or later to better match available national data. Among cases' mothers, 12.5% (n = 39; 95% CI: 9.0-16.7) indicated they sought medical advice for fertility issues with the index child, which is similar to ever use of fertility services in the U.S. in 2002 (15.3% for those with at least one child). However, we found a higher proportion of cases conceived through ART (n =14, 4.8%; 95%CI: 2.7%-7.8%) compared to an overall prevalence of 1.2% in the US in 2002-2006. We further assessed the proportion of cases born after ART within strata of low birth weight, a strong risk factor for HB, and plurality to assess if the increase in ART use was due to either plurality or birth weight. Within each plurality strata the proportion of ART cases was similar for cases overall, those with low birth weight, and those with very low birth weight. Overall, the proportion of ART cases who were singletons did not appear to be elevated (observed: n = 4, 1.4% (95% CI: 0.4-3.5), national: 0.6%). We did, however, observe a higher than expected proportion of twins conceived through ART compared to national data (observed: n = 8, 50% (95% CI: 24.7-75.3), national: 16.4%). In addition, we found a high proportion of cases with reported BWS who were conceived through ART (n = 4, 33.3%; 95% CI: 9.9-65.1)). In conclusion, we found a possible excess of children conceived through ART among cases diagnosed with hepatoblastoma and within those cases diagnosed with BWS. This excess appears to be limited to twin pregnancies. These findings provide tentative evidence that there is an excess of children conceived through ART among HB cases.
Original languageEnglish (US)
StatePublished - Apr 17 2010


  • Beckwith Wiedemann syndrome
  • birth weight
  • cancer research
  • case control study
  • child
  • childhood cancer
  • confidence interval
  • female
  • fertility
  • hepatoblastoma
  • human
  • infertility
  • learning
  • low birth weight
  • methylation
  • mother
  • national health organization
  • oncology
  • parent
  • prevalence
  • risk
  • risk factor
  • technology
  • twin pregnancy
  • twins
  • United States
  • very low birth weight


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